Особенности диагностического поиска при синдроме Черджа–Стросс. Клинический случай

Кузнецова А.С., Мельник С.И., Гаврилов П.В., Сергеев К.В., Трусова О.В. Особенности диагностического поиска при синдроме Черджа–Стросс. Клинический случай. Педиатрия. Consilium Medicum.
2023;2:198–204. DOI: 10.26442/26586630.2023.2.202314

© ООО «КОНСИЛИУМ МЕДИКУМ», 2023 г.

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Kuznetsova AS, Melnik SI, Gavrilov PV, Sergeyev KV, Trusova OV. Features of diagnostic search in Churg–Strauss syndrome. Case report. Pediatrics. Consilium Medicum. 2023;2:198–204. DOI: 10.26442/26586630.2023.2.202314

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Аннотация

Синдром Черджа–Стросс (СЧС) – редкий системный васкулит, поражающий сосуды мелкого калибра, клинически проявляется в виде аллергического риносинусита, бронхиальной астмы (БА), эозинофилии, легочных инфильтратов. Причиной поздней диагностики является отсутствие специфических особенностей дебюта, постепенная манифестация клинических проявлений, типичная для системных заболеваний. Клинический случай описан с целью усовершенствовать знания врачей-педиатров и пульмонологов о роли диагностического поиска при СЧС для маршрутизации, наблюдения пациента и коррекции терапии. Мальчик 17 лет с диагнозом БА поступил в отделение пульмонологии для уточнения, верификации диагноза, подбора терапии. Проведены инструментальное и лабораторное исследования: компьютерная томография (КТ) показала «мигрирующие» инфильтраты обоих легких, что может соответствовать зонам эозинофильной инфильтрации; КТ полисинусита, полипоза воздухоносных полостей носа; функция внешнего дыхания – без патологии; антитела к миелопероксидазе – 14 Ед/мл (норма меньше 5), антитела к протеиназе-3 – 30 Ед/мл (норма меньше 5), антитела к сахаромицетам – 6,3–7,2 Ед/мл, эозинофилия – 12%, (абс. 840 кл/л), при повторном анализе – результат отрицательный. На основании критериев Американской коллегии ревматологов: БА, эозинофилия выше 10% (абс. 840 кл/л), риносинусит, мигрирующие инфильтрации, по данным мультиспиральной КТ органов грудной клетки установлен диагноз СЧС. Выписан на комбинированную терапию с высокими возрастными дозировками ингаляционных глюкокортикостероидов, на фоне которой состояние стабилизировалось, потребности в системных глюкокортикостероидах не возникло. Описанный клинический случай наглядно демонстрирует трудность и длительность диагностики СЧС из-за стадийности течения заболевания, частого (33% случаев) отсутствия лабораторных признаков васкулита (антитела к сахаромицетам – отрицательно) и значительной трудности верификации диагноза на ранних этапах. Диагноз предусматривает комплексный и междисциплинарный подходы к диагностике и лечению.

Ключевые слова: клинический случай, синдром Черджа–Стросс, васкулит, бронхиальная астма, педиатрия

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Churg–Strauss syndrome is a rare systemic vasculitis affecting small-caliber vessels, which has characteristic clinical manifestations in the forms of allergic rhinosinusitis, bronchial asthma (BA), eosinophilia, and pulmonary infiltrates. The reason for late diagnostics is gradual manifestation of symptoms, that typical of systemic diseases. Improving the knowledge of pediatricians, pulmonologists about the role of diagnostic search in Churg–Strauss syndrome for further routing, monitoring and correction of patient therapy. A 17-year-old boy with BA was admitted to the pulmonology department for clarification, verification of the diagnosis, and selection of therapy. Instrumental and laboratory examination was carried out: on the computer tomography (CT) scan of the thoracic organs "migrating" infiltration of both lungs, based on laboratory data may correspond to eosinophilic zones. On the CT scan of the nasal sinuses polysinusitis, polyposis; external respiration function (spirography) – no pathology; myeloperoxidase-antineutrophil cytoplasmic antibodies (ANCA) – 14 U/ml (normal <5), anti-proteinase 3 anti-neutrophil ANCA – 30 U/ml (normal <5), anti-Saccharomyces cerevisiae antibodies – 6.3–7.2 U/ml, eosinophilia (12%, abs 840 c). The result of the level of antibodies was negative later. Churg–Strauss syndrome was based on the American College of Rheumatology criteria: BA, eosinophilia >10% (abs 840), rhinosinusitis, migrating infiltrations on CT scan. He was discharged for asthma combination therapy with high age doses of inhaled corticosteroids. After treatment his condition improved, there was no need for systemic corticosteroids. The described clinical case clearly demonstrates the difficulty and duration of diagnosing Churg–Strauss syndrome due to the stage-by-stage course of the disease, the frequent (33% of cases) absence of laboratory evidence of vasculitis (ANCA-neg.) significant difficulty of verifying the diagnosis at early stages. The diagnosis provides for a comprehensive and interdisciplinary approach to diagnosis and treatment.

Keywords: clinical case, Churg–Strauss syndrome, vasculitis, bronchial asthma, pediatrics

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Список литературы

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2005;52(9):2926-35. DOI:10.1002/art.21250
12. Lyons PA, Peters JE, Alberici F, et al. Genome-wide association study of eosinophilic granulomatosis with polyangiitis reveals genomic loci stratified by ANCA status. Nat Commun. 2019;10(1):5120. DOI:10.1038/s41467-019-12515-9
13. Schmitt WH, Csernok E, Kobayashi S, et al. Churg–Strauss syndrome: serum markers of lymphocyte activation and endothelial damage. Arthritis Rheum. 1998;41(3):445-52. DOI:10.1002/1529-0131(199803)41:3<445::AID-ART10>3.0.CO;2-3
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22. Clutterbuck EJ, Evans DJ, Pusey CD. Renal involvement in Churg–Strauss syndrome. Nephrol Dial Transplant. 1990;5(3):161-7. DOI:10.1093/ndt/5.3.161
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24. Kawakami T, Soma Y, Kawasaki K, et al. Initial cutaneous manifestations consistent with mononeuropathy multiplex in Churg–Strauss syndrome. Arch Dermatol. 2005;141(7):873-8. DOI:10.1001/archderm.141.7.873
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28. Szczeklik W, Sokołowska B, Mastalerz L, et al. Pulmonary findings in Churg–Strauss syndrome in chest X-rays and high resolution computed tomography at the time of initial diagnosis. Clin Rheumatol. 2010;29(10):1127-34. DOI:10.1007/s10067-010-1530-3
29. Бекетова Т.В., Волков М.Ю. Международные рекомендации по диагностике и лечению эозинофильного гранулематоза с полиангиитом – 2015. Научно-практическая ревматология. 2016;54(2):129-37 [Beketova TV, Volkov MYu. The 2015 International guidelines for the diagnosis and treatment of eosinophilic granulomatosis with polyangiitis. Nauchno-Prakticheskaya Revmatologiya=Rheumatology Science and Practice. 2016;54(2):129-37 (in Russian)]. DOI:10.14412/1995-4484-2016-129-137
30. Marchand E, Reynaud-Gaubert M, Lauque D, et al. Idiopathic chronic eosinophilic pneumonia. A clinical and follow-up study of 62 cases. The Groupe d'Etudes et de Recherche sur les Maladies “Orphelines” Pulmonaires (GERM “O”P). Medicine (Baltimore). 1998;77(5):299-312. DOI:10.1097/00005792-199809000-00001
31. Seccia V, Baldini C, Latorre M, et al. Focus on the Involvement of the Nose and Paranasal Sinuses in Eosinophilic Granulomatosis with Polyangiitis (Churg–Strauss Syndrome): Nasal Cytology Reveals Infiltration of Eosinophils as a Very Common Feature. Int Arch Allergy Immunol. 2018;175(1-2):61-9. DOI:10.1159/000484602
32. Ameratunga R, Steele R. Eosinophilic granulomatosis with polyangiitis (Churg–Strauss vasculitis) presenting as Samter's triad. J Allergy Clin Immunol Pract. 2018;6(1):280-2. DOI:10.1016/j.jaip.2017.07.006
33. Akuthota P, Weller PF. Eosinophilic pneumonias. Clin Microbiol Rev. 2012;25(4):649-60. DOI:10.1128/CMR.00025-12
34. Valent P, Klion AD, Horny HP, et al. Contemporary consensus proposal on criteria and classification of eosinophilic disorders and related syndromes. J Allergy Clin Immunol. 2012;130(3):607-12.e9. DOI:10.1016/j.jaci.2012.02.019
35. Loricera J, Blanco R, Ortiz-Sanjuán F, et al. Single-organ cutaneous small-vessel vasculitis according to the 2012 revised International Chapel Hill Consensus Conference Nomenclature of Vasculitides: a study of 60 patients from a series of 766 cutaneous vasculitis cases. Rheumatology (Oxford). 2015;54(1):77-82. DOI:10.1093/rheumatology/keu295
36. Klion AD, Ackerman SJ, Bochner BS. Contributions of Eosinophils to Human Health and Disease. Annu Rev Pathol. 2020;15:179-209.
DOI:10.1146/annurev-pathmechdis-012419-032756
37. Guillevin L, Pagnoux C, Seror R, et al. The Five-Factor Score revisited: assessment of prognoses of systemic necrotizing vasculitides based on the French Vasculitis Study Group (FVSG) cohort. Medicine (Baltimore). 2011;90(1):19‑27. DOI:10.1097/MD.0b013e318205a4c6
38. Sinico RA, Bottero P. Churg–Strauss angiitis. Best Pract Res Clin Rheumatol. 2009;23(3):355-66. DOI:10.1016/j.berh.2009.02.004
39. Gayraud M, Guillevin L, le Toumelin P, et al. Long-term followup of polyarteritis nodosa, microscopic polyangiitis, and Churg–Strauss syndrome: analysis of four prospective trials including 278 patients. Arthritis Rheum. 2001;44(3):666‑75. DOI:10.1002/1529-0131(200103)44:3<666::AID-ANR116>3.0.CO;2-A
40. Chung KF, Wenzel SE, Brozek JL, et al. International ERS/ATS guidelines on definition, evaluation and treatment of severe asthma. Eur Respir J. 2014;43(2):343-73. DOI:10.1183/09031936.00202013
41. Birck R, Schmitt WH, Kaelsch IA, van der Woude FJ. Serial ANCA determinations for monitoring disease activity in patients with ANCA-associated vasculitis: systematic review. Am J Kidney Dis. 2006;47(1):15-23. DOI:10.1053/j.ajkd.2005.09.022

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1. Watts RA, Lane S, Scott DG. What is known about the epidemiology of the vasculitides? Best Pract Res Clin Rheumatol. 2005;19(2):191-207. DOI:10.1016/j.berh.2004.11.006
2. Ntatsaki E, Watts RA, Scott DG. Epidemiology of ANCA-associated vasculitis. Rheum Dis Clin North Am. 2010;36(3):447-61. DOI:10.1016/j.rdc.2010.04.002
3. Masi AT, Hunder GG, Lie JT, et al. The American College of Rheumatology 1990 criteria for the classification of Churg–Strauss syndrome (allergic granulomatosis and angiitis). Arthritis Rheum. 1990;33(8):1094-100. DOI:10.1002/art.1780330806
4. Jennette JC, Falk RJ, Bacon PA, et al. 2012 Revised international Chapel Hill consensus conference nomenclature of vasculitides. Arthritis Rheum. 2013;65(1):1‑11. DOI:10.1002/art.37715
5. Zwerina J, Eger G, Englbrecht M, et al. Churg–Strauss syndrome in childhood: a systematic literature review and clinical comparison with adult patients. Semin Arthritis Rheum. 2009;39(2):108-15. DOI:10.1016/j.semarthrit.2008.05.004
6. Gendelman S, Zeft A, Spalding SJ. Childhood-onset eosinophilic granulomatosis with polyangiitis (formerly Churg–Strauss syndrome): a contemporary single-center cohort. J Rheumatol. 2013;40(6):929-35. DOI:10.3899/jrheum.120808
7. Iudici M, Puéchal X, Pagnoux C, et al. Brief Report: Childhood-Onset Systemic Necrotizing Vasculitides: Long-Term Data from the French Vasculitis Study Group Registry. Arthritis Rheumatol. 2015;67(7):1959-65. DOI:10.1002/art.39122
8. Vaglio A, Martorana D, Maggiore U, et al. HLA-DRB4 as a genetic risk factor for Churg–Strauss syndrome. Arthritis Rheum. 2007;56(9):3159-66. DOI:10.1002/art.22834
9. Conron M, Beynon HL. Churg–Strauss syndrome. Thorax. 2000;55(10):870-7. DOI:10.1136/thorax.55.10.870
10. Sada KE, Amano K, Uehara R, et al. A nationwide survey on the epidemiology and clinical features of eosinophilic granulomatosis with polyangiitis (Churg–Strauss) in Japan. Mod Rheumatol. 2014;24(4):640-4. DOI:10.3109/14397595.2013.857582
11. Sinico RA, Di Toma L, Maggiore U, et al. Prevalence and clinical significance of antineutrophil cytoplasmic antibodies in Churg–Strauss syndrome. Arthritis Rheum.
2005;52(9):2926-35. DOI:10.1002/art.21250
12. Lyons PA, Peters JE, Alberici F, et al. Genome-wide association study of eosinophilic granulomatosis with polyangiitis reveals genomic loci stratified by ANCA status. Nat Commun. 2019;10(1):5120. DOI:10.1038/s41467-019-12515-9
13. Schmitt WH, Csernok E, Kobayashi S, et al. Churg–Strauss syndrome: serum markers of lymphocyte activation and endothelial damage. Arthritis Rheum. 1998;41(3):445-52. DOI:10.1002/1529-0131(199803)41:3<445::AID-ART10>3.0.CO;2-3
14. Hellmich B, Ehlers S, Csernok E, Gross WL. Update on the pathogenesis of Churg–Strauss syndrome. Clin Exp Rheumatol. 2003;21(6 Suppl. 32):S69-77.
15. Vaglio A, Martorana D, Maggiore U, et al. HLA-DRB4 as a genetic risk factor for Churg–Strauss syndrome. Arthritis Rheum. 2007;56(9):3159-66. DOI:10.1002/art.22834
16. Exley AR, Bacon PA, Luqmani RA, et al. Development and initial validation of the vasculitis damage index for the standardized clinical assessment of damage in the systemic vasculitides. Arthritis Rheum. 1997;40(2):371-80. DOI:10.1002/art.1780400222
17. Nataraja A, Mukhtyar C, Hellmich B, et al. Outpatient assessment of systemic vasculitis. Best Pract Res Clin Rheumatol. 2007;21(4):713-32. DOI:10.1016/j.berh.2007.01.004
18. Churg A. Recent advances in the diagnosis of Churg–Strauss syndrome. Mod Pathol. 2001;14(12):1284-93. DOI:10.1038/modpathol.3880475
19. Katzenstein AL. Diagnostic features and differential diagnosis of Churg–Strauss syndrome in the lung. A review. Am J Clin Pathol. 2000;114(5):767-72.
DOI:10.1309/F3FW-J8EB-X913-G1RJ
20. Lie JT. Illustrated histopathologic classification criteria for selected vasculitis syndromes. American College of Rheumatology Subcommittee on Classification of Vasculitis. Arthritis Rheum. 1990;33(8):1074-87. DOI:10.1002/art.1780330804
21. Hueto-Perez-de-Heredia JJ, Dominguez-del-Valle FJ, Garcia E, et al. Chronic eosinophilic pneumonia as a presenting feature of Churg–Strauss syndrome. Eur Respir J. 1994;7(5):1006-8.
22. Clutterbuck EJ, Evans DJ, Pusey CD. Renal involvement in Churg–Strauss syndrome. Nephrol Dial Transplant. 1990;5(3):161-7. DOI:10.1093/ndt/5.3.161
23. Neumann T, Manger B, Schmid M, et al. Cardiac involvement in Churg–Strauss syndrome: impact of endomyocarditis. Medicine (Baltimore). 2009;88(4):236-43. DOI:10.1097/MD.0b013e3181af35a5
24. Kawakami T, Soma Y, Kawasaki K, et al. Initial cutaneous manifestations consistent with mononeuropathy multiplex in Churg–Strauss syndrome. Arch Dermatol. 2005;141(7):873-8. DOI:10.1001/archderm.141.7.873
25. Lanham JG, Elkon KB, Pusey CD, Hughes GR. Systemic vasculitis with asthma and eosinophilia: a clinical approach to the Churg–Strauss syndrome. Medicine (Baltimore). 1984;63(2):65-81. DOI:10.1097/00005792-198403000-00001
26. Pagnoux C, Guillevin L. Churg–Strauss syndrome: evidence for disease subtypes? Curr Opin Rheumatol. 2010;22(1):21-8. DOI:10.1097/BOR.0b013e328333390b
27. Grayson PC, Ponte C, Suppiah R, et al. 2022 American College of Rheumatology/European Alliance of Associations for Rheumatology Classification Criteria for Eosinophilic Granulomatosis With Polyangiitis. Arthrit Rheumatol. 2022;74(3):386-92. DOI:10.1002/art.41982
28. Szczeklik W, Sokołowska B, Mastalerz L, et al. Pulmonary findings in Churg–Strauss syndrome in chest X-rays and high resolution computed tomography at the time of initial diagnosis. Clin Rheumatol. 2010;29(10):1127-34. DOI:10.1007/s10067-010-1530-3
29. Beketova TV, Volkov MYu. The 2015 International guidelines for the diagnosis and treatment of eosinophilic granulomatosis with polyangiitis. Nauchno-Prakticheskaya Revmatologiya=Rheumatology Science and Practice. 2016;54(2):129-37 (in Russian). DOI:10.14412/1995-4484-2016-129-137
30. Marchand E, Reynaud-Gaubert M, Lauque D, et al. Idiopathic chronic eosinophilic pneumonia. A clinical and follow-up study of 62 cases. The Groupe d'Etudes et de Recherche sur les Maladies “Orphelines” Pulmonaires (GERM “O”P). Medicine (Baltimore). 1998;77(5):299-312. DOI:10.1097/00005792-199809000-00001
31. Seccia V, Baldini C, Latorre M, et al. Focus on the Involvement of the Nose and Paranasal Sinuses in Eosinophilic Granulomatosis with Polyangiitis (Churg–Strauss Syndrome): Nasal Cytology Reveals Infiltration of Eosinophils as a Very Common Feature. Int Arch Allergy Immunol. 2018;175(1-2):61-9. DOI:10.1159/000484602
32. Ameratunga R, Steele R. Eosinophilic granulomatosis with polyangiitis (Churg–Strauss vasculitis) presenting as Samter's triad. J Allergy Clin Immunol Pract. 2018;6(1):280-2. DOI:10.1016/j.jaip.2017.07.006
33. Akuthota P, Weller PF. Eosinophilic pneumonias. Clin Microbiol Rev. 2012;25(4):649-60. DOI:10.1128/CMR.00025-12
34. Valent P, Klion AD, Horny HP, et al. Contemporary consensus proposal on criteria and classification of eosinophilic disorders and related syndromes. J Allergy Clin Immunol. 2012;130(3):607-12.e9. DOI:10.1016/j.jaci.2012.02.019
35. Loricera J, Blanco R, Ortiz-Sanjuán F, et al. Single-organ cutaneous small-vessel vasculitis according to the 2012 revised International Chapel Hill Consensus Conference Nomenclature of Vasculitides: a study of 60 patients from a series of 766 cutaneous vasculitis cases. Rheumatology (Oxford). 2015;54(1):77-82. DOI:10.1093/rheumatology/keu295
36. Klion AD, Ackerman SJ, Bochner BS. Contributions of Eosinophils to Human Health and Disease. Annu Rev Pathol. 2020;15:179-209.
DOI:10.1146/annurev-pathmechdis-012419-032756
37. Guillevin L, Pagnoux C, Seror R, et al. The Five-Factor Score revisited: assessment of prognoses of systemic necrotizing vasculitides based on the French Vasculitis Study Group (FVSG) cohort. Medicine (Baltimore). 2011;90(1):19‑27. DOI:10.1097/MD.0b013e318205a4c6
38. Sinico RA, Bottero P. Churg–Strauss angiitis. Best Pract Res Clin Rheumatol. 2009;23(3):355-66. DOI:10.1016/j.berh.2009.02.004
39. Gayraud M, Guillevin L, le Toumelin P, et al. Long-term followup of polyarteritis nodosa, microscopic polyangiitis, and Churg–Strauss syndrome: analysis of four prospective trials including 278 patients. Arthritis Rheum. 2001;44(3):666‑75. DOI:10.1002/1529-0131(200103)44:3<666::AID-ANR116>3.0.CO;2-A
40. Chung KF, Wenzel SE, Brozek JL, et al. International ERS/ATS guidelines on definition, evaluation and treatment of severe asthma. Eur Respir J. 2014;43(2):343-73. DOI:10.1183/09031936.00202013
41. Birck R, Schmitt WH, Kaelsch IA, van der Woude FJ. Serial ANCA determinations for monitoring disease activity in patients with ANCA-associated vasculitis: systematic review. Am J Kidney Dis. 2006;47(1):15-23. DOI:10.1053/j.ajkd.2005.09.022

Авторы

А.С. Кузнецова*¹, С.И. Мельник^2,3, П.В. Гаврилов², К.В. Сергеев², О.В. Трусова¹

¹ФГБОУ ВО «Первый Санкт-Петербургский государственный медицинский университет им. акад. И.П. Павлова» Минздрава России, Санкт-Петербург, Россия;
²ФГБУ «Санкт-Петербургский научно-исследовательский институт фтизиопульмонологии» Минздрава России, Санкт-Петербург, Россия;
³ФГБОУ ВО «Северо-Западный государственный медицинский университет им. И.И. Мечникова» Минздрава России, Санкт-Петербург, Россия
*alexmorozova29@mail.ru

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Alexandra S. Kuznetsova*¹, Svetlana I. Melnik^2,3, Pavel V. Gavrilov², Kirill V. Sergeyev², Olga V. Trusova¹

¹Pavlov First Saint Petersburg State Medical University, Saint Petersburg, Russia;
²Saint Petersburg State Research Institute of Phthisiopulmonology, Saint Petersburg, Russia;
³Mechnikov North-Western State Medical University, Saint Petersburg, Russia
*alexmorozova29@mail.ru

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